Chorea is a rare adverse effect of phenytoin and is common in children, with widely variable clinical presentations. This article reported a case of an elderly woman presented chorea without nystagmus, ataxia, dysarthria and other typical vestibular-cerebellar symptoms, who took compound theophylline and ephedrine contained phenytoin to treat asthma for 1 year. The serum phenytoin concentrations were at toxic levels and chorea disappeared within 3 days after discontinuation of the drug. The clinical features of previously reported cases of phenytoin-induced chorea were also summarized.